普肯耶细胞功能障碍导致共济失调小鼠睡眠紊乱

IF 4 3区 医学 Q2 CELL BIOLOGY Disease Models & Mechanisms Pub Date : 2024-06-01 Epub Date: 2024-06-12 DOI:10.1242/dmm.050379
Luis E Salazar Leon, Amanda M Brown, Heet Kaku, Roy V Sillitoe
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引用次数: 0

摘要

浦肯野细胞功能障碍会扰乱运动并导致共济失调等疾病。最近的证据表明,浦肯野细胞功能障碍也可能改变睡眠调节。在这里,我们利用通过沉默普肯耶细胞神经递质而产生的共济失调小鼠模型(L7Cre;Vgatfx/fx)来更好地了解小脑功能障碍如何影响睡眠生理。我们将分析重点放在睡眠结构和皮层电图(ECoG)模式上,因为它们与提取睡眠期间的生理测量值息息相关。我们发现,突变小鼠的昼夜节律活动没有改变,但它们的睡眠参数和心电图模式有所改变。L7Cre;Vgatfx/fx突变小鼠的觉醒和快速眼动(REM)睡眠减少,而非快速眼动(NREM)睡眠增加。突变体的快速眼动睡眠潜伏期延长,这在人类共济失调患者身上也能观察到。心电信号的频谱分析显示,定义睡眠的不同频段的功率分布发生了变化。因此,浦肯野细胞功能障碍可能会影响共济失调患者的觉醒和不同睡眠阶段的平衡。我们的研究结果表明小脑功能障碍与睡眠紊乱之间存在联系,并强调了研究睡眠障碍中小脑回路功能的重要性。
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Purkinje cell dysfunction causes disrupted sleep in ataxic mice.

Purkinje cell dysfunction disrupts movement and causes disorders such as ataxia. Recent evidence suggests that Purkinje cell dysfunction may also alter sleep regulation. Here, we used an ataxic mouse model generated by silencing Purkinje cell neurotransmission (L7Cre;Vgatfx/fx) to better understand how cerebellar dysfunction impacts sleep physiology. We focused our analysis on sleep architecture and electrocorticography (ECoG) patterns based on their relevance to extracting physiological measurements during sleep. We found that circadian activity was unaltered in the mutant mice, although their sleep parameters and ECoG patterns were modified. The L7Cre;Vgatfx/fx mutant mice had decreased wakefulness and rapid eye movement (REM) sleep, whereas non-REM sleep was increased. The mutants had an extended latency to REM sleep, which is also observed in human patients with ataxia. Spectral analysis of ECoG signals revealed alterations in the power distribution across different frequency bands defining sleep. Therefore, Purkinje cell dysfunction may influence wakefulness and equilibrium of distinct sleep stages in ataxia. Our findings posit a connection between cerebellar dysfunction and disrupted sleep and underscore the importance of examining cerebellar circuit function in sleep disorders.

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来源期刊
Disease Models & Mechanisms
Disease Models & Mechanisms 医学-病理学
CiteScore
6.60
自引率
7.00%
发文量
203
审稿时长
6-12 weeks
期刊介绍: Disease Models & Mechanisms (DMM) is an online Open Access journal focusing on the use of model systems to better understand, diagnose and treat human disease.
期刊最新文献
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